Cauliflower Ear: A Rare Manifestation of Phaeohyphomycosis

Sir,

Phaeohyphomycosis is a subcutaneous mycosis caused by a variety of dematiaceous fungi. The causative organisms enter the dermis and subcutis by traumatic inoculation. The commonest skin manifestation is a cystic abscess in the extremities, systemic involvement being rare. Occurrence of phaeohyphomycosis in the ear following ear piercing is a rare complication which has not yet been reported in the literature.

A 46-year-old female presented with painful swelling of the right ear since last 2 months, following ear piercing with thorn of a plant. The lesion initially started as a swelling at the site of ear piercing in the helix, then it progressively involved the antihelix, triangular fossa, scaphoid fossa, and concha of the right ear occluding the external auditory meatus. There was no history of similar lesions elsewhere in the skin and the patient was not a diabetic.

On examination, there was a multiloculated, tender, cystic swelling in the left ear involving concha obscuring the normal morphology of the external ear [Figure 1a]. On aspiration, there was a seropurulent aspirate from the swelling [Figure 1b]. The aspirated pus was sent for potassium hydroxide (KOH) mount, Gram staining, fungal and bacterial cultures. Gram staining was negative for bacteria and the bacterial culture did not reveal any growth. KOH mount also failed to demonstrate fungal elements. Fungal culture at 37°C showed cream colored colony which later became brownish black [Figure 2a and b]. Lactophenol cotton blue (LCB) mount showed hyaline septate hyphae with arthrospores of varying sizes and shapes and budding blastoconidia within the hyphae [Figure 2c]. The hyphae also showed smooth-walled elliptical conidia [Figure 2d] which were consistent with the features of Aureobasidium pullulans. Biochemical tests or polymerase chain reaction sequencing for confirmation of species of fungus was not done because of the nonavailability.

Figure 1.

(a) Cystic swelling in right ear. (b) Seropurulent aspirate from the swelling

Figure 2.

(a) Culture on Sabouraud Dextrose Peptone Agar (SDA) showing brownish black fungal colony with slimy whitish surface on the top. (b) Culture on the reverse showing brownish black colony. (c) Fungal hyphae of Aureobasidium showing variable sized arthrospores and budding blastospores within the hyphae (black arrows) in LCB mount. (d) Hyphae with smooth-walled elliptical conidia (black arrows) of Aureobasidium pullulans in LCB mount

Skin biopsy from right ear showed collections of pigmented hyphae and spores in the dermis [Figure 3a-c]. Periodic acid Schiff (PAS) staining of the skin biopsy section also confirmed fungal spores and hyphae in the tissue section [Figure 3d]. The diagnosis of phaeohyphomycosis was made and the patient was given itraconazole 200 mg twice daily for 3 months. The swelling gradually diminished with residual “cauliflower ear” deformity of the external ear [Figure 4].

Figure 3.

(a) Scanning view showing pigmented fungal hyphae (black arrow) in the dermis (H and E, ×40). (b) Skin biopsy showing normal epidermis with upper dermis showing fungal colonies (black arrow) (H and E, ×100). (c) High power view of skin biopsy showing pigmented fungal hyphae and spores (H and E, ×400). (d) PAS stained section shows pink colored fungal spores (H and E, ×400)

Figure 4.

Residual cauliflower ear deformity after itraconazole therapy

Phaeohyphomycosis is caused by a heterogeneous group of fungi producing pigmented hyphae and yeast-like cells in the tissues. It usually presents as solitary nodule, abscess, or large cystic lesion in the extremities.[1] Innocuous pustule in the hand, nodules on the surgical scar, erythematous nodules, and tumid plaques over the face and hand and erythematous indurated nodules in the lower limbs with a discharging sinus are the few rare presentations of phaeohyphomycosis. Dissemination of organism is uncommon and there is no tendency toward lymphatic spread.[2]

Aureobasidium pullulans are saprophytes that are ubiquitous in the environment. There are few case reports of phaeohyphomycosis caused by Aureobasidium pullulans in both immunocompetent and immunosuppressed patients. Two cases of multiple cystic lesions involving the extremities with underlying immunosuppression were reported by Ponnuswamy et al.,[3] whereas a foot abscess caused by Aureobasidium pullulans in an immunocompetent patient was reported by Fernandes et al.[4]

Cauliflower ear commonly occurs following hematoma auris[5] and the invasive treatment modalities used for “pseudocyst of the ear,” had not yet been reported due to phaeohyphomycosis. This case is reported for its unique presentation of phaeohyphomycosis in ear and its sequelae “cauliflower ear” following indigenous ear piercing technique with the thorn of a plant.

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Conflicts of interest

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